Abstract
Acromegaly is associated with an increased incidence of neoplasia thought to be related in part to tonic increases in circulating levels of insulin-like growth factor-1 (IGF-1). Hemangiopericytomas, particularly those occurring in soft tissues, are known to possess IGF receptors. These tumors often behave aggressively and can be recalcitrant to surgery and radiation therapy. A 49-yr-old man presented with an erosive, midline scalp mass and cutis gyrata. Further diagnostic imaging and endocrinologic analysis confirmed two diagnoses: a hemangiopericytoma and acromegaly associated with an intrasellar pituitary tumor. Both the hemangiopericytoma and the pituitary adenoma were surgically resected; Western blot analysis confirmed the presence of IGF-1 receptors in the hemangiopericytoma. Two years later, the patient underwent resection of a right frontal hemangiopericytoma with an identical histologic phenotype and receptor positivity for IGF-1. The occurrence of a central nervous system hemangiopericytoma in patient with acromegaly is rare. Growth and recurrence of hemangiopericytoma may have been fostered by the presence of IGF-1 receptors. Paracrine mechanisms related to IGF-1 may have contributed to its pathogenesis and growth. The presence of IGF-1 receptors in hemangiopericytomas may have treatment implications for additional adjuvant therapies.
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References
Cushing H. Partial hypophysectomy for acromegaly: with remarks on the function of the hypophysis. Ann Surg 50:1002–1017, 1909.
Laws ER Jr, Piepgras DG, Randall RV. Neurosurgical management of acromegaly: results in 82 patients treated between 1972 and 1977. J Neurosurg 50:454–461, 1979.
Thapar K, Kovacs KT, Stefaneau L, Scheithauer BW, Horvath E, Lloyd RV, Li J, Laws ER Jr. Antiproliferative effect of the somatostatin analogue octreotide on growth hormone-producing pituitary tumors: results of a multicenter randomized trial. Mayo Clin Proc 72:893–900, 1997.
Steiner L, Payne BR, Prasad D, Lindquiest C, Steiner M. Gamma surgery in cerebral lesions, malformations, tumors, and functional disorders. In: Schmidek HH, ed. Operative neurosurgical techniques. Philadelphia: W.B. Saunders, 2000; 688, 689.
Kao PC, Laws ER Jr, Zimmerman D. Somatomedin C/insulin-like growth factor I levels after treatment of acromegaly. Ann Clin Lab Sci 22:95–99, 1992.
Giustina A, Barkan A, Casanueva FF, Cavagnini F, Frohman L, Ho K, Veldhuis J, Wass J, Von Werder K, Melmed S. Criteria for cure of acromegaly: a consensus statement. J Clin Endocrinol Metab 85(2):526–529, 2000.
Cushing H, Eisenhardt L. Angioblastic and transitional angioblastic meningioma. In: Meningiomas: their classification, regional behavior, life history, and surgical end results. Springfield, IL: Charles C Thomas, 1938; 42–47.
Begg CF, Garret R. Hemangiopericytoma occurring in the meninges. Cancer 7:602–606, 1954.
Stout AP, Murray MR. Hemangiopericytoma: a vascular tumor featuring Zimmerman’s pericyte. Ann Surg 116:26–33, 1942.
Kleihues P, Cavenee WK, eds. Hemangiopericytoma. In: Pathology and genetics of tumours of the nervous system. Lyon, France: IARC, 2000; 190–192.
Goellner JR, Laws ER Jr, Soule EH, Okazaki H. Hemangiopericytoma of the meninges: Mayo Clinic experience. Am J Clin Pathol 70:375–380, 1978.
Guthrie BL. Meningeal hemangiopericytomas. In Laws ER Jr, Kaye AH, eds. Brain tumors. New York: Churchill Livingstone, 1995; 705–710.
Guthrie BL, Ebersold MJ, Scheithauer BW. Meningeal hemangiopericytoma: histopathological features, treatment, and long term of 44 cases. Neurosurgery 25:514–522, 1989.
Wegmann W, Vonesch HJ, Kamber J, Kiss D. Recurrent and metastasizing hemangiopericytoma of the meninges with neoplastic hypoglycemia. Schweiz Med Wochenschr 124:146–151, 1994.
Lantos PL, Vandenburg SR, Kleihues P, Lopes MB. Hemangiopericytomas. In: Graham DI, Lantos PL, eds. Greenfield’s neuropathology. 6th ed. London: Oxford University Press, 1997; 745–748.
Glick RP, Gettleman R, Patel K, Lakshman R, Tsibris JC. Insulin and insulin-like growth factor I in brain tumors: binding and in vitro effects. Neurosurgery 24:791–797, 1989.
Kurihara M, Tokunaga Y, Tsutsumi K, Kawaguchi T, Shigematsu K, Niwa M, Mori K. Characterization of insulin-like growth factor I and epidermal growth factor receptors in meningiomas. J Neurosurg 71:538–544, 1989.
Chung J, Henry RR. Mechanisms of tumorinduced hypoglycemia with intraabdominal hemangiopericytoma. J Clin Endocrinol Metab 81:919–925, 1996.
Cotterill AM, Holly JM, Davies SC, Coulson VJ, Price PA, Wass JA. The insulin-like growth factors and their binding proteins in a case of non-islet-cell tumour-associated hypoglycemia. J Endocrinol 131:303–311, 1991.
Lowe WL Jr, Roberts CT Jr, LeRoith D, Rojeski MT, Merimee TJ, Fui ST, Keen H, Arnold D, Mersey J, Gluzman S. Insulin-like growth factor-II in nonislet cell tumors associated with hypoglycemia: increased levels of messenger ribonucleic acid. J Clin Endocrinol Metab 69:1153–1159, 1989.
Pavelic K, Cabrijan T, Hrascan R, et al. Molecular pathology of hemangiopericytomas accompanied by severe hypoglycemia: oncogenes, tumor-suppressor genes and the insulin-like growth factor family. J Cancer Res Clin Oncol 124:307–314, 1998.
Sohda T, Yun K. Insulin-like growth factor II expression in primary hemangiopericytoma and its metastasis to the liver accompanied by hypoglycemia. Hum Pathol 27:858–861, 1996.
Rotwein P, Burgess SK, Milbrandt JD, Krause JE. Differential expression of insulin-like growth factor genes in rat central nervous system. Proc Natl Acad Sci USA 85:265–269, 1988.
Bailey P, Cushing H, Eisenhardt L. Angioblastic meningiomas. Arch Pathol 6:953–990, 1928.
Grunenberger F, Bachellier P, Chenard MP, Massard G, Caraman PL, Perrin E, Zapf J, Jaeck D, Schlienger JL. Hepatic and pulmonary metastases from a meningeal hemangiopericytoma and severe hypoglycemia due to abnormal secretion of insulin-like growth factor: a case report. Cancer 85:2245–2248, 1999.
Davis DH, Laws ER Jr, Ilstrup DM, Speed JK, Caruso M, Shaw EG, Abboud CF, Scheithauer BW, Root LM, Schleck C. Results of surgical treatment for growth hormone-secreting pituitary adenomas. J Neurosurg 79:70–75, 1993.
Ross DA, Wilson CB. Results of transphenoidal microsurgery for growth hormone-secreting pituitary adenoma in a series of 214 patients. J Neurosurg 68:854–867, 1988.
Sharma S, Longo WE, Baniadam B, Vernava AM. Colorectal manifestations of endocrine disease. Dis Colon Rectum 38:318–323, 1995.
Popovic V, Damjanovic S, Micic D, Nesovic M, Djurovic M, Petakov M, Obradovic S, Zoric S, Simic M, Penezic Z, Marinkoviv. Increased incidence of neoplasia in patients with pituitary adenomas. The Pituitary Study Group. Clin Endocrinol Oxf 49:441–445, 1998.
Orme SM, McNally RJ, Cartright RA, Belchetz PE. Mortality and cancer incidence in acromegaly: a retrospective cohort study. United Kingdom Acromegaly Study Group. J Clin Endocrinol Metab 83:2730–2734, 1998.
Laws ER Jr. Acromegaly and gigantism. In: Wilkins RH, Rengachary SS, eds. Neurosurgery. New York: McGraw-Hill, 1996; 1317–1320.
Balkany C, Cushing GW. An association between acromegaly and thyroid carcinoma. Thyroid 5:47–50, 1995.
Sanno N, Teramoto A, Osamura RY, Genka S, Katakami H, Jin L, Lloyd RV, Kovacs K. A growth hormone-releasing hormone-producing pancreatic islet cell tumor metastastic to the pituitary is associated with pituitary somatotroph hyperplasia and acromegaly. J Clin Endocrinol Metab 82:2731–2737, 1997.
Cannavo S, Curto L, Fazio R, Paterniti S, Blandino A, Marafioti T, Trimarchi F. Coexistence of growth hormone-secreting pituitary adenoma and intracranial meningioma. J Endocrinol Invest 16:703–708, 1993.
Blevins LS Jr, Hall GS, Madoff DH, Laws ER Jr, Wand GS. Case report: acromegaly and Cushing’s disease in a patient with synchronous pituitary adenomas. Am J Med Sci 304:294–297, 1992.
Kolawole TM, Al-Orainy IA, Patel PJ, Fathuddin S. Cutis verticis gyrata: its computed tomographic demonstration in acromegaly. Eur J Radiol 27:145–148, 1998.
Hultberg BM, Haselbacher G, Nielsen FC, Wulff BS, Gammeltoft S. Gene expression of insulin-like growth factor II in human intracranial meningioma. Cancer 72:3282–3286, 1993.
Lichtor T, Kurpakus MA, Gurney ME. Expression of insulin-like growth factors and their receptors in human meningiomas. J Neurooncol 17:183–190, 1993.
Daughaday WH, Kapadia M. Significance of abnormal serum binding of insulin-like growth factor II in the development of hypoglycemia in patients with non-islet-cell tumors. Proc. Natl Acad Sci USA 86:6778–6782, 1989.
Thapar K, Kovacs K, Stefaneanu L, Scheithauer B, Killinger DW, Lloyd RV, Smyth HS, Barr A, Thorner MO, Gaylinn B, Laws ER Jr. Overexpression of the growth-hromone-releasing gene in acromegaly-associated pituitary tumors: an event associated with neoplastic progression and aggressive behavior. Am J Pathol 151(3):769–784, 1997.
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Elias, W.J., Hussaini, I.M., Chadduck, J.B. et al. Hemangiopericytoma in the setting of acromegaly. Endocr Pathol 13, 251–261 (2002). https://doi.org/10.1385/EP:13:3:251
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DOI: https://doi.org/10.1385/EP:13:3:251